Whipple's Disease Diagnosed During Anti-tumor Necrosis Factor Alpha Treatment

Two Case Reports and Review of the Literature

Jose M. Ramos; Francisco Pasquau; Nora Galipienso; Beatriz Valero; Angela Navarro; Agustín Martinez; José Rosas; Ana Gutiérrez; Rosario Sanchez-Martínez

Disclosures

J Med Case Reports. 2015;9(165)

Consent

Written informed consent was obtained from the patients for publication of this case report and any accompanying images. A copy of the written consent is available for review by the Editor-in-Chief of this journal.

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Abbreviations
AS: Ankylosis spondylitis, CT: Computed tomography, ESR: Erythrocyte sedimentation rate, HC: Hydroxychlorochine, m: month, MCV: Mean corpuscular volume, MTX: Methotrexate, NR: Not reported, PAS: Periodic acid–Schiff, PR: Present report, PCR: Polymerase chain reaction, RA: Rheumatoid arthritis, SA: Spondyloarthropathy (seronegative chronic polyarthritis), SD: Still's diseases, SXT: Cotrimoxazole, rRNA: ribosomal ribonucleic acid, TNF-α: Tumor necrosis factor alpha, w: week, WBC: White blood cell, WD: Whipple's disease, y: year

Acknowledgements
We should like to acknowledge the personnel from the Department of Internal Medicine Rheumatology and Gastroenterology of the Hospital General Universitario de Alicante and the Hospital Marina Baixa, Villajoyosa, Alicante (Spain) for attending to the patients.

Table 1. Features of the patients' diagnosis of Whipple's disease after tumor necrosis factor alpha antagonist initiation
Authors/country/language of publication	Age/sex	Underlying disease previous WD diagnosis/time with that diagnosis	TNF-α drugs and immunosuppressive treatment	Start of acute symptoms after TNF-α blockade	Symptoms after TNF-α blockade	Organ involvement	Histological diagnosis ^a	Result of PCR for Tropheryma whipplei	Treatment for WD	Outcome
Kneitz et al. (2005) [4]/Germany/English	34y/M	SD	Methotrexate + infliximab (2w)	2w	Weight loss, erythema nodosum, diarrhea, lymph node enlargement, and a sigmoidovesical fistula	Skin, gastroduodenitis and sigmoidovesical	Positive in fistula and lymph nodes	Positive in lymph node, small bowel and sigmoidovesical fistula	SXT (12m)	Resolved within 1 year
Kremer et al. (2008) [7]/Germany/German	47y/M	SA/4y	Leflunomide + adalimumab		Fever, weight loss, and severe arthralgia		Positive in duodenal tissue	Positive in duodenal tissue	SXT
Spoerl et al. (2009) [5]/Switzerland/English	64y/M	SA/5 + 3y	Etanercept	4m	Lethargy, night sweats, weight loss	Gastrointestinal and vertebral	Positive in duodenal tissue and vertebral tissue	Positive in gastric and vertebral tissue	Ceftriaxone (2w), then, SXT (2y), (12m) then doxycycline + HC (18m)	No relapses after 34-month follow-up
Ahmadi-Simab et al. (2009) [8]/Germany/German	33y/F	AS/8y	Etanercept	12m	Diarrhea, abdominal pain and weight loss		Negative in duodenal tissue	Positive in duodenal biopsy	Ceftriaxone (2w) after SXT
Hoppé et al. (2010) [6]/France/English	67y/F	SA/7y	Infliximab (4m)	4m	Chest pain, dyspnea, polyarthritis		Positive in duodenal tissue	Positive in duodenal tissue. Negative in blood and saliva	Ceftriaxone (2w), then doxycycline (24m)	Resolved within 15d. No relapses after 71-month follow-up
Hoppé et al. (2010) [6]/France/English	40y/M	AS/1y	Infliximab (18m) then etanercept (7m) then adalimumab (1m)	26m	Diarrhea, weight loss, fever, arthralgia	Widespread ileocolitis, gastroduodenitis, meningitis	Positive in duodenal tissue	Positive in saliva, feces and cerebrospinal fluid. Negative in blood	Doxycycline, HC, and SXT (15d), then SXT was replaced by sulfasalazine 4g/day	Resolved within 2 months. No relapses after 17-month follow-up
Hoppé et al. (2010) [6]/France/English	60y/M	AS/8y	Etanercept (9m)	9m	Fever, night sweats, polyarthritis, chest pain	Pericarditis, mediastinal and abdominal lymphadenopathy, duodenitis	Negative in duodenal tissue	Positive in the duodenal sample, saliva, feces, and lymph nodes	Doxycycline + HC (15m)	Resolved within 7d. No relapses after 30-month follow-up
Hoppé et al. (2010) [6]/France/English	47y/M	RA/17y	Infliximab (36m), then etanercept (42m), then adalimumab (6m), then rituximab, abatacept then infliximab (1m)	85m	Fever, night sweats, weight loss, polyarthritis, radio carpal arthropathy. transient diplopia, myalgia, subacute depression	Pericarditis, abdominal lymphadenopathy, duodenitis		Positive PCR assay in the duodenal sample, blood, saliva, and feces. Negative PCR assay in cerebrospinal fluid	Doxycycline + HC (UT)	Resolved within 2m No relapses after 15-month follow-up
Hoppé et al. (2010) [6]/France/English	38y/M	AS/9y	Etanercept (2m)	9m	Diarrhea, abdominal pain. weight loss, blurred vision	Hemorrhagic gastroduodenitis, hemorrhagic colitis, mediastinal and cervical lymphadenopathy, splenomegaly, meningitis	Positive in duodenum and colon	Positive PCR assay in blood, saliva, cerebrospinal fluid, and feces	Doxycycline 200mg/day. HC 600mg/day. (UT)	Resolved within 3w. No relapses after 13-month follow-up
Hmamouchi et al. (2010) [9]/France/English	35y/M	AS/4y	Etanercept	4m	Gingival nodule, purpura, abdominal pain diarrhea, fatigue, weight loss	Gingiva and colitis	Positive in colon tissue	Positive in blood, stool, and cerebrospinal fluid	SXT (NR)	Resolved within 3 months
Daïen et al. (2010) [10]/France/English	70y/M	SA/27y	Etanercept (18m)	NR	Fever and dyspnea	Endocarditis of aortic valve	Negative in duodenal tissue and, aortic valve	Positive in aortic valve	Doxycycline HC + SXT (18m)	Resolved. No relapses after 21-month follow-up
Gaddy et al., (2012) [12]/USA/English	46y/M	AS/10y	Prednisone + MTX + infliximab (24m), then adalimumab	24m	Fever, migratory arthritis, weight loss, diarrhea	Gastroenteritis	Positive in duodenal tissue	Positive in blood and duodenal tissue	Ceftriaxone (x2w) after SXT	NR
Sparsa et al. (2013) [11]/France/French	53y/M	SA	Etanercept (3m), then adalimumab (6m)	9m	Polyarthralgia peripheric, arthritis metarsophalangeal		Positive in duodenal tissue biopsy	Positive in saliva and duodenal tissue	Doxycycline + HC (18m)	Resolved within 3 weeks. No relapses after 18-month follow-up
Sparsa et al. (2013) [11]/France/French	42y/M	SA/2m	Adalimumab (9m) and then etanercept (4m)	13m	Polyarthralgia peripheric		Positive in duodenal and gastric tissue	Positive in saliva and duodenal tissue	Doxycycline + HC and then doxycycline alone	Resolved within 3 weeks. No relapses after 17- month follow-up
Present report	58y/M	Psoriatic spondyloarthropathy/14y	Adalimumab (4m), then etanercept (8m), infliximab (2m), tocilizumab (21m), golimumab (1m)	36m	Abdominal pain, diarrhea and weight loss	Duodenitis and abdominal lymph nodes	Positive in duodenal tissue	Not done	Ceftriaxone (14d), then SXT during (UT)	Resolved within 3 weeks. No relapses after 9-month follow-up.
Present report	78y/M	RA/19y	Infliximab (6m), etanercept (6m), rituximab (3m), leflunomide (24m)	24m	Abdominal pain, diarrhea and weight loss	Duodenitis, and pericardial effusion	Positive in duodenal tissue	Positive in duodenal biopsy. Negative in cerebrospinal fluid	Ceftriaxone (14d), then SXT for (12m) then doxycycline + HC	Resolved within 3 weeks. No relapses after 18-month follow-up

UT) Patient still under treatment
TNF-α tumor necrosis factor alpha, WD Whipple's disease, PCR polymerase chain reaction, y year, SD Still's disease, w week, SXT cotrimoxazole, m month, SA spondyloarthropathy (seronegative chronic polyarthritis), HC hydroxychlorochine, AS ankylosis spondylitis, RA rheumatoid arthritis, NR not reported, MTX methotrexate
^aHistological diagnosis = intracellular bacilli on periodic acid-Schiff stain

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Whipple's Disease Diagnosed During Anti-tumor Necrosis Factor Alpha Treatment

Two Case Reports and Review of the Literature

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Authors and Disclosures

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